EUROPEAN JOURNAL OF PHYSICAL AND REHABILITATION MEDICINE - riabilitazione … A... · 2014. 7. 16. · Setting: Outpatient clinic. Population: Singlecase. Methods: We developed a

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A pilot rehabilitation program based on cueing for Freezing

of Speech

Roberto ERRO, Maria Rosaria TEDESCHI, Carmine VITALE, Stefania

BUONOCORE, Giuseppe OREFICE

Eur J Phys Rehabil Med 2014 Apr 03 [Epub ahead of print]

EUROPEAN JOURNAL OF PHYSICAL AND REHABILITATION

MEDICINERivista di Medicina Fisica e Riabilitativa dopo Eventi Patologici

pISSN 1973-9087 - eISSN 1973-9095

Article type: Case Report

The online version of this article is located at http://www.minervamedica.it

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A (single case) rehabilitation program based on cueing for Freezing of Speech

Roberto Erro1, Maria Rosaria Tedeschi2, Carmine Vitale3,4, Stefania Buonocore2, Giuseppe Orefice1.

1. Department of Neurological Science, University of Naples Federico II2. Department of Logopedics and Phoniatrics, University of Naples Federico II

3. IDC Hermitage, Capodimonte4. University of Naples Parthenope

Congress: noneFunding: none: Conflicts of interest: noneAcknowledgements: none

Correspondance to:

Dr. Roberto Erro, MDDepartment of Neurological Science, University of Naples Federico IIVia Pansini 5, 80131, Napoli (IT) email: [email protected]: (+39)0817462670fax: (+39)0815466596

ABSTRACT

Background: Freezing of speech (FoS) and other repetitive speech behaviours can frequently occur in

parkinsonian syndromes, worsening the efficacy of language functioning, hampering social interactions, and

thus reducing quality of life. Pharmacological treatment are ineffective and other interventions have not

specifically developed so far.

Aim: To test the efficacy on a pilot rehabilitation program for freezing of speech based on cueing.

Setting: Outpatient clinic.

Population: Singlecase.

Methods: We developed a pilot rehabilitation program based on visual, auditory and sensory cueing. The

patient underwent the protocol for a 6month period (1hour sessions, 3 times weekly). Perceptual

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4


analyses of his speech were performed before and after the rehabilitation program by to different blinded

reviewers.

Results: There has been a reduction of FoS and other iterative speech episodes. Intelligibility also improved

according to both the patient and his relatives.

Conclusion: Our preliminary results show that visual, auditory and sensory cueing can be effective to

prevent and overcome FoS episodes. Possible underlying mechanisms of the improvement are discussed.

Clinical Rehabilitation Impact: Rehabilitation programs based on cueing should be considered and offered

in Parkinsonian patients exhibiting FoS or other iterative speech phenomena.

Keywords: Parkinson’s disease, Rehabilitation, Speech therapy, freezing, freezing of speech, voice

therapy

TEXT

Introduction

Freezing phenomenon is a common motor disturbance in patients with parkinsonism [13]. It can be

frequently seen in Parkinson’s disease (PD) as well as in other “pure” freezing disorders [15]. Freezing

phenomena are sudden, lasting seconds, episodes that break the normal motion, inhibiting patients to

perform an automatic motor skill [1,2]. Being most frequently reported to affect gait, freezing has also been

described in association with speech, writing and brushing teeth [3].

Most of the evidences on the freezing phenomenon come from research on freezing of Gait (FoG). FoG

highly impairs mobility and reduces quality of life [6,7]. Diagnostic criteria for freezing behaviours other

than FoG do not exist, and, as such, it is difficult to know their true prevalence and burden among

parkinsonian patients. It is likely that freezing of speech (FoS) can worsen quality of life, reducing verbal

output and hampering social interactions, as it is seen in patients with hearing loss [8].



4

Previous studies have defined FoS as a brief, episodic absence or marked reduction of forward

progression of the speech, despite the intention to speak, bearing resemblance with FoG [1]. Other authors

have used the term repetitive speech phenomena, acknowledging that they could be speech equivalents

of the freezing phenomenon [9].

The freezing phenomenon is variable in nature and this is partly related to the influence of sensory and

cognitive inputs [10]. Although the underlying pathophysiology remains unknown, it has been suggested

that freezing is a sensorimotor phenomenon with hypersensitivity to visual stimulation [10]. Moreover,

strategies based on sensory, visual or auditory cueing have been demonstrated to be really effective on

FoG [11,12]. Whether external cueing may improve such freezing phenomenon as FoS, has never been

investigated.

Here, we report the efficacy of a pilot rehabilitation program based on cueing to improve FoS and other

iterative language behaviours in a patient suffering from Pure Akinesia with Freezing Gait (PAFG).

Case report

Patient

A 62yearold man was admitted in our department because of a speech problem that started 11 months

earlier with the inability to start speaking. His speech disturbances progressively worsened and 8 months

after onset, he started to complain also gait difficulties. He felt as his feet were ‘‘glued to the ground’’,

when starting gait or while turning. Neurological examination revealed no abnormalities other than FOG

and speech abnormalities (as described below). There were no further extrapyramidal features. Mini

Mental State Examination score was 27 out of 30 indicating a normal cognitive status. Brain Magnetic

Resonance Imaging was unremarkable, while [I123]FPCIT brain SPECT disclosed a symmetrical

reduced uptake in both putamen and caudate, and 18[F]FDG PET showed a selective hypometabolism of

the midbrain, supporting the clinical diagnosis of PAFG [13] and excluding other disorders potentially

responsible for the symptoms. LDopa, rasagiline and duloxetine at adequate dosages were ineffective to

improve both FoG and FoS. Due to the reported efficacy of cueing strategies in alleviating FoG in



4

parkinsonian patients, we developed a rehabilitation program based on sensory, visual and auditory cueing

as reported below. The patient underwent the protocol for a 6month period (1hour sessions, 3 times

weekly). A speech evaluation was performed before (T0=baseline) and after (T1=6 months) the

rehabilitation program and data compared between T0 and T1. At these times, the FogQuest (i.e. a

validated tool to score FoG severity) was also administrated, its value considered a score of disease

progression [14]. During the overall evaluation period, patient was at a stable regimen of duloxetine (60 mg

daily). Moreover, he refused any rehabilitation program for his gait disturbances.

Speech Evaluation

During the test procedure, our patient was seated in a quiet room and received detailed instructions on how

to perform each test. He was asked to speak as slowly and distinctively as possible. Four speech

production tasks were tested: naming, reading, repetition and spontaneous speech, while comprehension

was assessed by Token Test [15]. Naming was evoked by showing patient 40 drawings (20 nouns, 20

action) [16]. Reading test required the patient to read aloud 10 increasing in length and complexity

sentences containing normal words [16]. Repetition was checked asking patient repeat aloud 20 words, 20

nonwords and 10 sentences of increasing length and complexity [16]. Patient was asked to produce

spontaneous speech during an extensive, semistructured interview with requests for both procedural and

personal informations (i.e. “Tell me how you would go about having a shower” and “Tell me what you

usually do on Sunday”). Spontaneous speech was digitally videorecorded. Two independent reviewers

(MRT, CV) blinded to the patient and to the time of assessment, screened the video for the appearance of

FoS and other repetitive speech phenomena. When there has been disagreement between the reviewers, a

third one (RE) was requested to assess the video. Given that a discrete definition of FoS does not exist, we

considered episodes of FoS those characterised by phonatory effort, associated with a break of normal

speech motion. Similar to the “trembling on place” seen in FoG, an acoustically identifiable iteration of

phonemes could be associated with FoS. Other iterative phenomena (palilalia, oral festination, etc.) were



4

defined according to the literature [17,18]. Three different 60s epochs were assessed, the results averaged

and are here provided as number of episodes/minute.

Speech Rehabilitation Program

We developed a rehabilitation program based on three types of cueing: auditory, visual and sensory.

The auditory level included exercises of reading and repetition of words increasing in length and

complexity, syllabling them in time with a metronome. Moreover, patient was requested to read and repeat

passages with homologous (nursery rhymes) or variable (poems) pauses.

The sensory level included exercises of reading and repetition of increasing in length and complexity words,

syllabling them in time with a touching stimulus (the trainer tapping on the patient’s leg or the patient tapping

on his own leg).

The visual level included both “silent” and “sonorous” exercises at the mirror, imitating the trainer or looking

at a drawing (fig. 1) in order to exaggerate oral and tongue movements able to produce different

phonemes. Moreover, patient was also requested to imagine the oral movement necessary for the

production of different phonemes to overcome FoS episodes.

Results

Data of the speech evaluation at both T0 and T1 are summarised in table 1.

No significant abnormalities were found in reading, naming, repetition and comprehension at both T0 and

T1. At baseline, 15 episodes/minute of palilalia, 2 episodes of echolalia and 6 of FoS were detected, while

other iterative speech behaviours were not present. After the rehabilitation program,

2 episodes of palilalia, 2 of echolalia and 1 of FoS were detected. Both the patient and his relatives

reported a subjective improvement with regards of speech intelligibility. At the baseline the NewFogQuest

score was 23, while at follow up was 30.

Discussion

The results of our pilot study show that a rehabilitation program based on cueing can reduce FoS and other

repetitive phenomena, and improve language functioning in a patient affected with PAFG.



4

The worsening of the index we used to score FoG severity in our patient, points the rehabilitation program

as causative of the improvement observed in the speech output.

PAFG is a rare syndrome that was first describedin the mid 1970s [19]. This condition was reported to

exhibit only freezing phenomenon, specifically frozen gait, micrographia and festinating speech [4,5,19].

The syndrome was initially thought to be a discrete clinical entityuntil when increasingdata emerged that it

may represent another phenotype for Progressive Supranuclear Palsy [19]. Of note, there is a close

resemblance between PAFG andPrimary Progressive Freezing Gait (PPFG), another rare disorder where

FoG is the sole neurological dysfunction with the exception of a varied degree of postural instability.

However, insight into the pathophysiology of PAFG and PPFG has come from imaging studies. In fact,

while PET/SPECT findings are abnormal in PAFG (as in our case) [13],PET and SPECT scanning usually

reveal no abnormalities in PPFG [19].

Although the effects of external cues on FoS have been never investigated so far, previous works based on

physical rehabilitation programs for FoG have found a beneficial effect of cueing in improving gait pattern

and overcoming freezing episodes in PD [11,12]. Main explanation for previous findings relies on the

hypothesis that cueing makes motor tasks less automatic, allowing patients to modify their motor strategies

(i.e. external cues may help patients to developed new attentional strategies and to switch from one

movement component of a sequence to the next) [12]. This would presume the involvement of different

cortical areas able to compensate the defective loop from basal ganglia to the supplementary motor area,

assumed to be putatively responsible for freezing occurrence [2,3,10].

In keeping with this suggestion, we suggest the same mechanism has led to the improvement observed in

our patient. Indeed, he referred that the internal visualisation of oral/tongue movement was the best strategy

to overcome FoS episodes. This could suggest that activation of visual and associative cortices may

bypass the trigger responsible for the FoS.



4

The increased attentional load in speaking could represent an alternative or additional mechanism. The

latter hypothesis has been in fact raised also for other types of speech therapy [20,21]. Singing therapy has

been suggested as an attentional strategy to consciously control voice production, with the advantage of

offering a rhythmic musical facilitation [20,21]. Over the years, a number of different speech rehabilitation

programs have been proposed. However, it should be noted that all these studies have focused on

impaired articulation and reduced loudness rather than on iterative speech behaviours [22]. As such,

comparison between our study and existing literature is not straightforward. All over, a recent review of

speech and language therapy techniques for speech problems in PD has shown that there is insufficient

evidence to support or refute the efficacy of any form of speech therapy over another to treat speech

disturbances in PD patients [22].

Although PAFG is rare, repetitive speech behaviour can be frequently seen in PD [9,18], the second most

common neurodegenerative disorder. Once again, it has been supposed that iterative speech phenomena in

PD are the result of a dysfunction of subcorticalcortical interplay. Whether FoS and palilalia (the two main

phenomena observed in our patient) reflect the same underlying pathophysiology is not known, but a role

played by subcortical structures can be suggested, given the results of both brain 18[F]FDG PET and

[I123]FPCIT SPECT. Whatever is the underlying mechanism, the rehabilitation program has been found

to have a positive effect on both phenomena, improving the general speech output and its intelligibility. We

acknowledge that our findings are based on perceptual evaluation of our patient’s speech while no

objective speech measurement (spectrogram) is provided. However, it should be noted that the reliability

of the spectrograph in evaluating “complex” speech disorders has not yet been tested. Moreover,

spectrogram analysis does not always reflect what is seen in daily life, and we deliberately focused on the

perceptual analysis that, in our opinion, can better reflect clinical relevant improvement. In fact, in our case

there was consistency between the subjective improvement reported by the patient (and his relatives) and

the rating obtained by two different (blinded) reviewers. Finally, we do not have data on longterm

outcome, and it is possible that with a further followup, longerstanding benefits of the rehabilitation



4

program will vanish. This would suggest that these procedures should be constantly applied and reinforced,

possibly being incorporated in patient’s lifestyle.

Conclusion

While preliminary, our results support the hypothesis that sensory cues are effective to improve FoS and

other iterative speech phenomena. This may encourage further research to develop better rehabilitation

protocols, in order to improve speech functioning and therefore social interactions and quality of life in

parkinsonian patients.

TITLE OF TABLES

Table 1. Speech performances before and after the rehabilitation program.

TITLE OF FIGURES

Figure 1. Example of the drawings used for the visual cueing.

REFERENCES

[1] Giladi N, McDermott MP, Fahn S, et al. Freezing of gait in PD: Prospective assessment in the

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[3] Giladi N, Fahn S. Freezing Phenomenon, the fifth cardinal sign of parkinsonism. Progress in

Alzheimer’s and Parkinson’s disease. 1998 Plenum Press, New York.

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a wide spectrum of PSP? In: Narabayashi H, Nagatsu T, Yanagisawa N, Mizuno Y, eds.: Advances in

neurology. New York: Raven Press, 1993;60:622–625.



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disease beyond its relationships with mobility and gait. Mov Disord 2007;22:2192–5.

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EUROPEAN JOURNAL OF PHYSICAL AND REHABILITATION MEDICINE - riabilitazione … A... · 2014. 7. 16. · Setting: Outpatient clinic. Population: Singlecase. Methods: We developed a